用计算方法分析三肽基肽酶 1 错义突变对神经细胞类脂膜炎的功能和结构影响。

IF 3.2 3区 医学 Q2 ENDOCRINOLOGY & METABOLISM Metabolic brain disease Pub Date : 2024-04-01 Epub Date: 2024-01-08 DOI:10.1007/s11011-024-01341-8
Priyanka K, Priya N Madhana, Rajalakshmanan Eswaramoorthy, Magesh Ramasamy
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引用次数: 0

摘要

神经细胞类脂膜脂质沉着病(NCLs)是一组严重的神经退行性疾病,多在婴儿期、晚期、少年期和成年期发病。它们的表型特征包括视力损伤、运动能力和认知功能减退,有时在发病初期就会死亡。在最近的研究中,NCL 被分为至少 14 个基因系列(CLN1-14)。CLN2基因编码三肽基肽酶1(TPP1),影响晚期婴儿发病形式。在本研究中,我们从各种数据库(ClinVar、UniProt、HGMD)中检索了 TPP1 蛋白的突变数据集进行筛选。通过计算方法列举了 56 个错义突变体,发现了重要的突变体(G475R 和 G501C),并与临床和文献数据进行了关联。为了了解蛋白质与配体之间的相互作用和动态模拟,我们启动了基于结构的筛选方法。对原生(3EDY)和突变体(G473R 和 G501C)结构与吉非罗齐(gem)进行了对接,后者能降低血脂水平,减少血液循环中甘油三酯的含量,控制高脂血症。原生体的相互作用得分为-5.57 kcal/mol,突变体的平均结合得分分别为-6.24(G473R)和-5.17(G501C)kcal/mol。最后,分子动力学模拟显示,在所有轨迹分析中,G473R 和 G501C 突变体具有更好的灵活性和稳定取向。因此,这项工作扩展了对导致 NCL 紊乱的突变体的功能和结构影响的理解。
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A computational approach to analyzing the functional and structural impacts of Tripeptidyl-Peptidase 1 missense mutations in neuronal ceroid lipofuscinosis.

Neuronal ceroid-lipofuscinosis (NCLs) are a group of severe neurodegenerative conditions, most likely present in infantile, late infantile, juvenile, and adult-onset forms. Their phenotypic characteristics comprise eyesight damage, reduced motor activity and cognitive function, and sometimes tend to die in the initial stage. In recent studies, NCLs have been categorized into at least 14 genetic collections (CLN1-14). CLN2 gene encodes Tripeptidyl peptidase 1 (TPP1), which affects late infantile-onset form. In this study, we retrieved a mutational dataset screening for TPP1 protein from various databases (ClinVar, UniProt, HGMD). Fifty-six missense mutants were enumerated with computational methods to perceive the significant mutants (G475R and G501C) and correlated with clinical and literature data. A structure-based screening method was initiated to understand protein-ligand interaction and dynamic simulation. The docking procedure was performed for the native (3EDY) and mutant (G473R and G501C) structures with Gemfibrozil (gem), which lowers the lipid level, decreases the triglycerides amount in the blood circulation, and controls hyperlipidemia. The Native had an interaction score of -5.57 kcal/mol, and the mutants had respective average binding scores of -6.24 (G473R) and - 5.17 (G501C) kcal/mol. Finally, molecular dynamics simulation showed that G473R and G501C mutants had better flexible and stable orientation in all trajectory analyses. Therefore, this work gives an extended understanding of both functional and structural levels of influence for the mutant form that leads to NCL disorder.

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来源期刊
Metabolic brain disease
Metabolic brain disease 医学-内分泌学与代谢
CiteScore
5.90
自引率
5.60%
发文量
248
审稿时长
6-12 weeks
期刊介绍: Metabolic Brain Disease serves as a forum for the publication of outstanding basic and clinical papers on all metabolic brain disease, including both human and animal studies. The journal publishes papers on the fundamental pathogenesis of these disorders and on related experimental and clinical techniques and methodologies. Metabolic Brain Disease is directed to physicians, neuroscientists, internists, psychiatrists, neurologists, pathologists, and others involved in the research and treatment of a broad range of metabolic brain disorders.
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